Promoting men’s awareness, self-examination, and help-seeking for testicular disorders: a systematic review of interventions

Background: Testicular cancer (TC) is among the most commonly diagnosed cancers in men aged 15–40 years. The incidence of TC is on the rise. Benign testicular disorders, such as testicular torsion and epididymitis, can lead to testicular ischemia, sepsis, and infertility if left untreated. This updated systematic review aims to evaluate the effectiveness of studies promoting men’s knowledge and awareness of testicular disorders and/or self-examination, behaviours and/or intentions to examine their testes, and help-seeking behaviours and/or intentions for testicular disorder symptoms. Methods: Academic Search Complete, Medline, CINAHL, PsycINFO, ERIC, the Cochrane Library, the World Health Organisation International Clinical Trials Registry Platform, and Clinicaltrials.gov were searched for studies published between April 2018 and August 2023. Methodological quality was assessed and results were synthesised meta-narratively. Results: Five studies were included. The majority of the reviewed interventions were successful in increasing men’s awareness of TC and self-examination, including a PowerPoint presentation, an online educational brochure, video-assisted teaching, a motivational video, and a virtual reality game. Only one study addressed help-seeking for testicular symptoms and promoted men’s awareness of benign as well as malignant testicular diseases. Conclusions: This review highlights the importance of evaluating innovative educational interventions aimed at younger men, whilst raising their awareness of testicular disorders and increasing their help-seeking intentions for testicular disorder symptoms. Given the lack of consensus around scheduled testicular self-examination among younger men, clinicians are encouraged to instruct men to familiarise themselves with the look and feel of their own testes and to seek timely medical attention for abnormalities. Registration: The protocol of the previous version of this review was registered with the International Prospective Register of Systematic Reviews (PROSPERO) under the registration number CRD42018093671.


Introduction
According to the National Cancer Institute, testicular cancer (TC) is among the most commonly diagnosed cancers in men aged 15 to 40 years. The incidence of TC has doubled globally over the past 40 years and is highest in Western and Northern European countries, Australia, and North America 1,2 . According to the National Cancer Registry Ireland, over 90% of TC cases and 85% of TC deaths in Ireland occur among men younger than 50 years. Furthermore, the incidence of TC in Ireland is increasing by 2.4% annually. A unilateral painless testicular mass is a classical sign of TC. Testicular pain, back pain, cough, haemoptysis, and headaches can be warning signs of metastatic TC 3,4 .
Benign testicular disorders (BTDs) can also have a negative impact on a man's health. Epididymo-orchitis, often contracted sexually by men younger than 50 years, is known to be the primary cause of acute scrotal pain and testicular enlargement. This infection can cause sepsis and infertility if not diagnosed and managed promptly 5 . Testicular torsion is characterised by severe scrotal pain, oedema, nausea, and vomiting, and can lead to testicular ischemia and necrosis if testicular perfusion is not restored within 6 hours from the onset of pain [5][6][7] . The severity of these conditions highlights the potential role of testicular awareness and testicular self-examination (TSE) in detecting TC as well as BTDs 8,9 .
A systematic review of 25 studies exploring men's awareness of TC and TSE found that men were unaware of TC risk factors, signs and symptoms, and treatments, and that very few reported performing TSE 10 . These findings were echoed by Roy and Casson, who explored the awareness, knowledge, and attitudes regarding TC and TSE of 150 men in Northern Ireland 11 . This study found that only 39% of participants correctly identified the TC at-risk age group, and only 17% were aware of TSE 11 . Sparse recent evidence exists in relation to BTD awareness. Saleem et al. explored men's awareness of BTDs in Pakistan and found that 78.8% of participants were unaware of the symptoms of BTDs, 73.6% reported that BTDs were considered taboo, and 29.8% did not intend to perform TSE 12 . Yap et al. surveyed Irish parents (n=242) about their awareness and help-seeking for testicular torsion 13 . This study found that parents who were aware of torsion were four times more likely to seek immediate help (OR, 4.2; 95% CI, 1.4-12.2; p<0.01) than those who lacked awareness. Moreover, participants who correctly identified the timeframe for help-seeking were three times more likely to seek immediate help than those who did not know the timeframe (OR, 3.0; 95% CI, 0.85-10.8; p=0.08) 13 .
There is no consensus regarding the effectiveness of monthly TSE in detecting testicular disorders early 14 , which resulted in different recommendations regarding this practice globally. For instance, the U.S. Preventive Services Task Force opposes this practice 15 , whereas Cancer Research UK and the Irish Cancer Society encourage men to check their testes and report any abnormalities to a healthcare professional. TSE proponents were critical of the decision made by U.S. Preventive Services Task Force and stated that TSE has potential benefits beyond the early detection of TC such as familiarising men with their own testes and helping detect TC and BTDs early 16 20 . Despite men's lack of awareness of BTDs and their intentions to delay help-seeking for symptoms of testicular disease, none of these reviews included studies that aimed at promoting men's awareness of BTDs and/or increasing their intentions to seek help for testicular symptoms. The present review builds upon the search, screening, and output from both reviews 19,20 . Of note, there is no gold standard for the frequency of updating structured reviews 21 . However, biennial review updates are recommended by the Cochrane Library.

Objectives
The aim of this updated systematic review is to evaluate the effectiveness of experimental studies promoting men's knowledge and awareness of testicular disorders and/or self-examination, behaviours and/or intentions to examine their testes, and help-seeking behaviours and/or intentions for testicular symptoms. The outcomes of this review are presented below using the PICOS (participants, interventions, comparisons, outcomes, and study design) framework: Outcomes: 1. The effect of intervention on men's knowledge and awareness of testicular disorders and/or self-examination, compared to baseline and/or control conditions (i.e., alternative intervention or no intervention).
2. The effect of intervention on men's behaviours and/or intentions to examine their testes, compared to baseline and/or control conditions (i.e., alternative intervention or no intervention).

UPDATE
3. The effect of intervention on men's help-seeking behaviours and/or intentions for testicular disorder symptoms.

Protocol and registration
This updated systematic review is reported using the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) checklist 22 . The review questions and methods were predetermined and were not amended during the review process. The protocol of the original review was registered with the International Prospective Register of Systematic Reviews (PROSPERO) under the registration number CRD42018093671.

Eligibility criteria
Studies were eligible for inclusion if they used any experimental design and were conducted among men who did not have a diagnosis of a testicular disorder. Studies addressing the review outcomes and studies evaluating the effect of intervention(s) compared to baseline and/or control conditions were included. The full inclusion criteria are reported in Table 1 using the PICOS framework.
Men with a diagnosis of a testicular disorder, studies with women only, studies with a paediatric population, and studies where findings from men, women, and/or paediatric populations are indistinguishable were excluded. Additionally, quantitative descriptive studies, qualitative studies, opinion papers, reviews of the literature, and conference abstracts were not eligible for inclusion. Theses and dissertations were also excluded because the merit of their use in systematic reviews is questionable 23 .

Information sources and search strategy
The following electronic databases were searched in August 2023: Academic Search Complete, Medline, CINAHL, PsycINFO, ERIC, and The Cochrane Library. In addition, eligible studies were sought from trial registries including the World Health Organisation International Clinical Trials Registry Platform (ICTRP) and Clinicaltrials.gov. Reference lists of eligible papers were also reviewed. As this is an updated version of a previous review, the search was limited to records published between April 2018 and August 2023.
The following keywords were searched based on title and abstract using Boolean operators "OR" and "AND": "testicular disease*" OR "testicular disorder*" OR "testicular cancer" OR "testicular neoplas*" OR "testicular tumor*" OR "testicular tumour*" OR "testicular malignan*" OR "benign testicular disorder*" OR "benign testicular disease*" OR "testicular torsion" OR epididymitis OR orchitis OR epididymoorchitis OR hydrocele OR varicocele OR spermatocele OR "testicular symptom*" OR "testicular pain" OR "testicular lump*" OR "testicular swelling" OR "scrot* symptom*" OR "scrot* pain" OR "scrot* lump*" OR "scrot* swelling" AND knowledge OR awareness OR practice* OR self-exam* OR "self exam*" OR feel* OR screen* OR "early detect*" OR help-seeking OR "help seeking" OR "help-seeking intention*" OR "help seeking intention*" OR "help-seeking behavior*" OR "help-seeking behaviour*" OR "help seeking behavior" OR "help seeking behaviour" AND intervention* OR inform* OR educat* OR "health education" OR "health promotion" OR trial* OR experiment* OR stud* OR program*.

Study selection and data extraction
Records identified from electronic databases and trial registries were exported to Covidence, an online software used to facilitate screening and data extraction. Duplicates were deleted automatically in Covidence.
All records were screened based on title and abstract. Following the exclusion of irrelevant records, the full text of potentially eligible studies was obtained for further screening. Title, abstract, and full-text screenings were conducted by two independent reviewers. Screening conflicts were resolved by consensus.
A standardised extraction table was used to extract data from experimental studies 19,20 . Data were extracted by one reviewer and cross-checked for accuracy by a second reviewer. The following data were extracted: author(s) and year; aim(s); country, setting and funding; participants; design and theoretical Table 1. Review inclusion criteria using the PICOS framework.

Participants
Adult men (aged 18+) without a diagnosis of a testicular disorder Interventions Educational/health promotion intervention/programme

Comparisons
The effect of intervention compared to baseline and/or control conditions i.e., alternative intervention(s) or no intervention Outcomes (i) Knowledge and awareness of testicular disorders and/or self-examination (ii) Behaviours and/or intentions to examine/feel own testes (iii) Help-seeking behaviours and/or intentions for testicular disorder symptoms

Study design
Any experimental design (i.e., randomised controlled trial, non-randomised controlled trial, pre-post study design with one or more groups, and post-test only study design with one or more groups) underpinning; intervention(s); outcome(s) and data collection; and findings presented according to the review questions.

Quality assessment
The methodological quality of the included studies was appraised using the Mixed Method Appraisal Tool (MMAT) which allows the appraisal of various study designs 24 . In the context of the present review, the quality of randomised controlled trials (RCT) and non-RCTs was appraised. Voting on each quality item was conducted on a "yes", "no", and "cannot tell" basis. Quality appraisal was conducted by one author and verified by a second author.

Data synthesis
A meta-analysis with summary measures of treatment effect using weighted/standard mean difference, risk/odds ratios, and 95% confidence was planned using RevMan 5, if the included studies were sufficiently homogenous. However, the included studies were heterogeneous in terms of intervention format, data collection, study design, and participant allocation; therefore, findings from the reviewed studies were synthesised meta-narratively.

Study selection
A total of 623 records were identified from electronic databases and clinical trial registries. No additional records were identified from reference list checks. Following the exclusion of duplicates, 608 records were screened based on title and abstract. Of those, 17 full-text articles were assessed for eligibility and 12 were excluded. Five studies were included in the present review. The full study selection process and reasons for exclusion are presented in Figure 1.

Study characteristics
Two studies were conducted in Turkey 25 27,29 , and the remaining study was an RCT 25 .

Quality assessment
All five studies had clear research questions and used appropriate data collection methods. The RCT met all the MMAT criteria 25 . As for non-RCTs (n=4), only two studies accounted for confounders and reported that participants were representative of the target population 26, 27 . It was unclear in two studies if measurements were appropriate regarding both, outcome and intervention 28,29 . While it was only unclear in one study if the intervention was administered as intended 28 . The four non-RCTs met the remaining MMAT criteria ( Table 2).

Synthesis of results
The full data extraction table and findings from individual studies are presented in Table 3. Behaviours and intentions to perform testicular selfexamination TSE behaviours and/or intentions were explored in four of the reviewed papers [25][26][27]29 . In the study by Akcali and Tastan, Table 2. Quality assessment using the mixed method appraisal tool.   only one participant in the intervention group and three in the control group reported practicing TSE 25 . This increased significantly to 82.4% (n=28) among the intervention group and 59.4% (n=19) among the control group at post-test (p<0.05) 25 . Over three quarters of participants (77.6%, n=38) in the study by Saab et al. reported that they intend to feel their testes at pre-test 27 . Of the 22.4% (n=11) participants who did not intent to feel their testes, 54.5% (n=6) reported feeling their testes at post-test 27  Help-seeking behaviours and intentions for testicular symptoms Only one study addressed men's help-seeking for testicular symptoms 27 . Help-seeking intention scores for testicular swelling and testicular lumps were 3.5/7 at pre-test which significantly increased to 3.8/7 immediately post-test for both symptoms (p<0.001 and p=0.003 respectively). Similarly, help-seeking intention scores for testicular pain were 3.2/7 at pre-test which increased significantly to 3.8/7 immediately post-test (p<0.001) 27 .

Summary of evidence
Five studies were included in this updated systematic review. Overall, the reviewed literature showed that there was an increase in men's awareness of TC and TSE and behaviours and intentions to perform TSE in response to various interventions, at least in the short-term. The included studies seldom addressed help-seeking behaviours and intentions for testicular symptoms. Indeed, only one study addressed this outcome and found a significant increase in intentions to seek help for symptoms of concern following a virtual reality game 27 .
Examples of interventions that successfully increased men's awareness of TC and TSE included: PowerPoint presentation underpinned by the Health Belief Model 25 , an online educational brochure also underpinned by the Health Belief Model 26 , video-assisted teaching 28 , a motivational video 29 , and a virtual reality game 27 . Of note, only Saab et al.'s study aimed to promote men's awareness of BTDs as opposed to only TC 27 . BTDs are more common than TC and a delay in help-seeking for benign testicular symptoms is also linked to negative health outcomes. For instance, a delay of more than 6 hours for pain caused by testicular torsion significantly reduces the chances of salvaging an ischemic testis 7 . Likewise, untreated epididymitis can lead to severe orchitis, sepsis, and in some cases irreversible infertility 5,6 .
As for TSE, a Cochrane review conducted by Ilic and Misso 14 found no definitive evidence regarding the risks and benefits of regular TSE; therefore it was recommended that at-risk groups, such as men with a family history of TC, undescended testis, or testicular atrophy, ought to be advised by their physician regarding the risks (e.g. false positives and concomitant anxiety) and benefits (e.g. early detection) of TSE. As a result, whether to conduct monthly TSE has been polarised into two competing positions. Since 2011, the U.S. Preventive Services Task Force "recommends against screening for testicular cancer in adolescent or adult men" 15  As stated, help-seeking was only addressed in one study 27 . A number of quantitative and qualitative descriptive studies found that men's intentions to seek help for testicular symptoms (e.g., lumpiness, swelling, and pain) are low [31][32][33] . Saab et al. conducted a qualitative descriptive study to explore men's (n=29) awareness of testicular disorders and intentions to seek help for testicular symptoms 33 . It was found that men lacked awareness of testicular disorders in general and BTDs in particular, as a result many reported that they would most likely delay help-seeking. In addition to lack of awareness, the following were identified as barriers to help-seeking: lack of familiarity with own testes, symptom misappraisal, low perceived risk of TC, embarrassment, fear, denial, false optimism, fatalism, machoism, stoicism, false reassurance by others, and healthcare system barriers such as access, cost and waiting time 33 . By contrast, the following were identified as facilitators to help-seeking: personal or family history of a testicular disease, inherent health-seeking drive, and access to support 33 34 . Thornton warned against the use of "cheeky" humour and puns as these can be potentially offensive and ineffective 35 . Another factor worth considering in health promotion intervention design and delivery is men's literacy and health literacy levels. A meta-narrative systematic review of 31 studies exploring men's information-seeking behaviours in relation to cancer prevention found that younger men and those with high literacy and health literacy levels were more likely to engage with information delivered using technological means 36 . By contrast, men who were older, belonged to ethnic minorities, and had low literacy and health literacy levels were more likely to engage with health information delivered by peers, physicians, and churches 36 .

Strengths and limitations
Rigour was ensured by systematically reporting this review using the PRISMA checklist. Moreover, a thorough search of electronic databases, trial registries, and reference lists was conducted, and records were independently screened by more than one reviewer to avoid omitting important records. However, the search was limited to records published between 2018 and 2023, which increases the risk of study selection bias, and only findings that were relevant to the review outcomes were discussed, which increases the risk of reporting bias. Due to heterogeneity, a meta-analysis was not plausible. Therefore, while promising interventions were identified and included in this updated review, definitive evidence regarding effectiveness cannot be determined.

Conclusions
The present updated review has implications for research and clinical practice, which should be considered carefully in light of the review limitations. From a research perspective, there is a need for population-level health interventions to promote men's awareness of testicular disorders. This could be achieved through considering the information needs and the preferred learning strategies of at-risk age groups, while accounting for sociodemographic variations within these groups 34 . It is also essential to factor in diseases other than TC (these were underexplored in the reviewed literature), and to conduct rigorous high-quality studies capturing the longitudinal impact of the interventions on behaviours and potentially on clinical outcomes such as stage at diagnosis, treatments received, and survival rates. Examples include but are not limited to: multimedia campaigns, virtual and augmented reality interventions, gaming technologies, mobile apps, and interactive websites.
The use of theory in intervention design and delivery is key, since interventions with a theoretical underpinning are more likely to achieve the desired outcomes, particularly when there is congruence between the assumptions of the theory and those of the proposed intervention 37 . An example is the Health Belief Model, which was used in two of the reviewed studies 25,26 . Another example is the Preconscious Awareness to Action Framework, a novel theoretical framework used by Saab et al. 27 to raise testicular awareness and promote early help-seeking for testicular symptoms.
From a practical standpoint, clinicians involved in health promotion are encouraged to direct men to resources where information on testicular disorders is freely and readily accessible. Given the scarcity of high-quality evidence to support scheduled TSE, lack of consensus regarding monthly TSE, clinicians should promote "testicular awareness" by encouraging men to become familiar with the look and feel of their own testes, to know which signs and symptoms to look for, and to seek prompt medical attention for symptoms of concern 8,30 .

Data availability
No data is associated with this article.